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Transplantation of induced neural stem cells (iNSCs) into chronically demyelinated corpus callosum ameliorates motor deficits

Multiple Sclerosis (MS) causes neurologic disability due to inflammation, demyelination, and neurodegeneration. Immunosuppressive treatments can modify the disease course but do not effectively promote remyelination or prevent long term neurodegeneration. As a novel approach to mitigate chronic stag...

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Autores principales: Sullivan, Genevieve M., Knutsen, Andrew K., Peruzzotti-Jametti, Luca, Korotcov, Alexandru, Bosomtwi, Asamoah, Dardzinski, Bernard J., Bernstock, Joshua D., Rizzi, Sandra, Edenhofer, Frank, Pluchino, Stefano, Armstrong, Regina C.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7285785/
https://www.ncbi.nlm.nih.gov/pubmed/32517808
http://dx.doi.org/10.1186/s40478-020-00960-3
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author Sullivan, Genevieve M.
Knutsen, Andrew K.
Peruzzotti-Jametti, Luca
Korotcov, Alexandru
Bosomtwi, Asamoah
Dardzinski, Bernard J.
Bernstock, Joshua D.
Rizzi, Sandra
Edenhofer, Frank
Pluchino, Stefano
Armstrong, Regina C.
author_facet Sullivan, Genevieve M.
Knutsen, Andrew K.
Peruzzotti-Jametti, Luca
Korotcov, Alexandru
Bosomtwi, Asamoah
Dardzinski, Bernard J.
Bernstock, Joshua D.
Rizzi, Sandra
Edenhofer, Frank
Pluchino, Stefano
Armstrong, Regina C.
author_sort Sullivan, Genevieve M.
collection PubMed
description Multiple Sclerosis (MS) causes neurologic disability due to inflammation, demyelination, and neurodegeneration. Immunosuppressive treatments can modify the disease course but do not effectively promote remyelination or prevent long term neurodegeneration. As a novel approach to mitigate chronic stage pathology, we tested transplantation of mouse induced neural stem cells (iNSCs) into the chronically demyelinated corpus callosum (CC) in adult mice. Male C57BL/6 mice fed 0.3% cuprizone for 12 weeks exhibited CC atrophy with chronic demyelination, astrogliosis, and microglial activation. Syngeneic iNSCs were transplanted into the CC after ending cuprizone and perfused for neuropathology 2 weeks later. Magnetic resonance imaging (MRI) sequences for magnetization transfer ratio (MTR), diffusion-weighted imaging (T2), and diffusion tensor imaging (DTI) quantified CC pathology in live mice before and after iNSC transplantation. Each MRI technique detected progressive CC pathology. Mice that received iNSCs had normalized DTI radial diffusivity, and reduced astrogliosis post-imaging. A motor skill task that engages the CC is Miss-step wheel running, which demonstrated functional deficits from cuprizone demyelination. Transplantation of iNSCs resulted in marked recovery of running velocity. Neuropathology after wheel running showed that iNSC grafts significantly increased host oligodendrocytes and proliferating oligodendrocyte progenitors, while modulating axon damage. Transplanted iNSCs differentiated along astrocyte and oligodendrocyte lineages, without myelinating, and many remained neural stem cells. Our findings demonstrate the applicability of neuroimaging and functional assessments for pre-clinical interventional trials during chronic demyelination and detect improved function from iNSC transplantation. Directly reprogramming fibroblasts into iNSCs facilitates the future translation towards exogenous autologous cell therapies.
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spelling pubmed-72857852020-06-11 Transplantation of induced neural stem cells (iNSCs) into chronically demyelinated corpus callosum ameliorates motor deficits Sullivan, Genevieve M. Knutsen, Andrew K. Peruzzotti-Jametti, Luca Korotcov, Alexandru Bosomtwi, Asamoah Dardzinski, Bernard J. Bernstock, Joshua D. Rizzi, Sandra Edenhofer, Frank Pluchino, Stefano Armstrong, Regina C. Acta Neuropathol Commun Research Multiple Sclerosis (MS) causes neurologic disability due to inflammation, demyelination, and neurodegeneration. Immunosuppressive treatments can modify the disease course but do not effectively promote remyelination or prevent long term neurodegeneration. As a novel approach to mitigate chronic stage pathology, we tested transplantation of mouse induced neural stem cells (iNSCs) into the chronically demyelinated corpus callosum (CC) in adult mice. Male C57BL/6 mice fed 0.3% cuprizone for 12 weeks exhibited CC atrophy with chronic demyelination, astrogliosis, and microglial activation. Syngeneic iNSCs were transplanted into the CC after ending cuprizone and perfused for neuropathology 2 weeks later. Magnetic resonance imaging (MRI) sequences for magnetization transfer ratio (MTR), diffusion-weighted imaging (T2), and diffusion tensor imaging (DTI) quantified CC pathology in live mice before and after iNSC transplantation. Each MRI technique detected progressive CC pathology. Mice that received iNSCs had normalized DTI radial diffusivity, and reduced astrogliosis post-imaging. A motor skill task that engages the CC is Miss-step wheel running, which demonstrated functional deficits from cuprizone demyelination. Transplantation of iNSCs resulted in marked recovery of running velocity. Neuropathology after wheel running showed that iNSC grafts significantly increased host oligodendrocytes and proliferating oligodendrocyte progenitors, while modulating axon damage. Transplanted iNSCs differentiated along astrocyte and oligodendrocyte lineages, without myelinating, and many remained neural stem cells. Our findings demonstrate the applicability of neuroimaging and functional assessments for pre-clinical interventional trials during chronic demyelination and detect improved function from iNSC transplantation. Directly reprogramming fibroblasts into iNSCs facilitates the future translation towards exogenous autologous cell therapies. BioMed Central 2020-06-09 /pmc/articles/PMC7285785/ /pubmed/32517808 http://dx.doi.org/10.1186/s40478-020-00960-3 Text en © The Author(s) 2020 Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Research
Sullivan, Genevieve M.
Knutsen, Andrew K.
Peruzzotti-Jametti, Luca
Korotcov, Alexandru
Bosomtwi, Asamoah
Dardzinski, Bernard J.
Bernstock, Joshua D.
Rizzi, Sandra
Edenhofer, Frank
Pluchino, Stefano
Armstrong, Regina C.
Transplantation of induced neural stem cells (iNSCs) into chronically demyelinated corpus callosum ameliorates motor deficits
title Transplantation of induced neural stem cells (iNSCs) into chronically demyelinated corpus callosum ameliorates motor deficits
title_full Transplantation of induced neural stem cells (iNSCs) into chronically demyelinated corpus callosum ameliorates motor deficits
title_fullStr Transplantation of induced neural stem cells (iNSCs) into chronically demyelinated corpus callosum ameliorates motor deficits
title_full_unstemmed Transplantation of induced neural stem cells (iNSCs) into chronically demyelinated corpus callosum ameliorates motor deficits
title_short Transplantation of induced neural stem cells (iNSCs) into chronically demyelinated corpus callosum ameliorates motor deficits
title_sort transplantation of induced neural stem cells (inscs) into chronically demyelinated corpus callosum ameliorates motor deficits
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7285785/
https://www.ncbi.nlm.nih.gov/pubmed/32517808
http://dx.doi.org/10.1186/s40478-020-00960-3
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